Epidemiology and clinical features of childhood-onset anti-neutrophil cytoplasmic antibody–associated vasculitis: a clinicopathological analysis View Full Text


Ontology type: schema:ScholarlyArticle     


Article Info

DATE

2019-05-10

AUTHORS

Daishi Hirano, Tomoaki Ishikawa, Aya Inaba, Mai Sato, Tomohiro Shinozaki, Kazumoto Iijima, Shuichi Ito

ABSTRACT

BackgroundThis study was performed to determine the clinical features and outcomes of childhood-onset anti-neutrophil cytoplasmic antibody (ANCA)–associated vasculitis (AAV), particularly microscopic polyangiitis (MPA).MethodsA retrospective Japanese multicenter study was performed in patients diagnosed with AAV before 16 years of age.ResultsOf 49 patients with AAV, 36 were female. The diagnoses were as follows: MPA (n = 38, 78%), granulomatosis with polyangiitis (GPA; n = 9, 18%), eosinophilic granulomatosis with polyangiitis (EGPA; n = 1, 2%), and other (n = 1, 2%). The median age at onset was 10.7 years, and median time to diagnosis was 2.0 months. Twenty-seven (55%) patients were identified through a school urinary screening program. Initial symptoms included fever and fatigue (45%), and renal (71%), pulmonary (29%), ocular (20%), and mucocutaneous involvement (22%). Although 27 (55%) patients achieved remission and none had died at the last follow-up, at least one recurrence occurred in 13 (48%) patients after a median of 48 months and was more common in patients with GPA (P < 0.01). After a median follow-up of 43 months, seven (14%) patients (all with MPA) progressed to end-stage renal disease (ESRD).ConclusionsChildhood-onset AAV has an estimated prevalence of 3.41–4.28 per million children and is characterized by female predominance and high frequency of detection in school urinary screening programs. More than 10% of patients with childhood-onset AAV still progress to ESRD without achieving remission. Histological chronicity is a factor associated with ESRD. More... »

PAGES

1425-1433

Identifiers

URI

http://scigraph.springernature.com/pub.10.1007/s00467-019-04228-4

DOI

http://dx.doi.org/10.1007/s00467-019-04228-4

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1114043935

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/31076873


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